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Nuclear Medicine & Radiation
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Eisenbach et al., J Nucl Med Radiat Ther 2015, 6:1
http://dx.doi.org/10.4172/2155-9619.1000209
Case Report
Open Access
The Role of Radiotherapy in the Treatment and Diagnosis of Pseudomalignant
Vertebral Haemangiomatosis: A Case Report
Colby Eisenbach*, Chase Hansen and Carlos Torres
TTUHSC School of Medicine, USA
*Corresponding
author: Colby Eisenbach, Medical Student, TTUHSC School of Medicine, USA, Tel: + 432-425-1855; E-mail: [email protected]
Received date: Jan 06, 2015, Accepted date: Jan 24, 2015, Publication date: Jan 27, 2015
Copyright: © 2015 Eisenbach C, et al. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits
unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
Abstract
Primary intraosseous haemangiomas represent benign endothelial neoplasms that are often discovered
incidentally in approximately 10 to 12 percent of the general population. Although bony haemangiomas can be found
in many regions of the appendicular and axial skeleton, most of which remain asymptomatic, those that arise in the
vertebral column have the potential to elicit pain and cause neurological deficits in certain patients. In fact, a subset
of primary vertebral intraosseous haemangiomas have been shown, albeit extremely rarely, to demonstrate
aggressive, destructive and even malignant behavior. From a clinical and therapeutic standpoint, it is essential to
recognize that such lesions possess certain radiological and nuclear imaging characteristics that, when observed in
conjunction with patient decline, correlate strongly with the atypical and aggressive neoplasms. Ultimately, such
lesions have the potential to give rise to an extremely uncommon, widespread, highly debilitating form of
haemangiomatous pathology, hereafter termed ''pseudomalignant haemangiomatosis''. Fortunately, treatment in the
form of directed radiotherapy has shown remarkable results in regards to symptomatic relief and complete
neurologic restoration.
Keywords: Primary intraosseous hemangioma; Radiotherapy for
hemangiomatosis; Radiation therapy; Hemangiomatosis of the spine;
Vertebral hemangioma; Hemangiomatosis; Aggressive hemangioma of
the spine
Background
First described by Virchow in 1867, primary intraosseous
haemangiomas of the spine are characterized by the benign
proliferation of the endothelial cells lining small blood vessels
traversing the trabecular bony matrix within the vertebrae [1-3].
Developing most commonly in the 4th and 5th decades of life, such
lesions are often discovered within the vertebral bodies, pedicles and
spinous processes of the thoracic and lumbar spine [3-5]. However,
contiguous extension and spread to the epidural space, lumbar
vertebrae, cervical vertebrae, femur, sternum, skull, and orbit have also
been documented in the literature [2-6]. By way of autopsy studies
performed by Topfer and Junghanns in 1928 and 1932 respectively,
the prevalence of such lesions within the general population is
predicted to be anywhere from 10 to 12 percent with a female to male
predominance of 9 to 2. Although the vast majority of vertebral
haemangiomas are asymptomatic and require no treatment, a subset of
these haemangiomatous disorders possess certain characteristics that
increase their likelihood of becoming symptomatic, destructive, and
even quasimetastatic in nature [7-11]. Due to the rarity and
unpredictability in the presentation and extent of these symptomatic
spinal haemangiomatous disorders, it should come as no surprise that
a wide array of treatment modalities have been developed to address
such cases including: decompressive laminectomy, short segment
posterior stabilization and fusion, intraarterial embolization,
vertebroplasty, kyphoplasty, intralesional ethanol injection and
radiotherapy [9,10,12-17]. Although no clear therapeutic protocol has
been shown to be superior in the treatment of incidental, isolated
J Nucl Med Radiat Ther
ISSN:2155-9619 JNMRT, an open access journal
vertebral haemangiomas, there is a prominent place for radiotherapy
in the treatment of symptomatic or ''atypical'' haemangiomatous
disease presentations [16-20]. Thus, in an effort to shed greater light
on the role of radiation therapy in the management of such
haemangiomatous variants and the multi-disciplinary approach that is
often required for successful treatment and symptom management, we
present a novel case of wide spread ''pseudomalignant'' spinal
haemangiomatosis characterized by multi-level spinal cord
compression, pathologic vertebral fractures, and appendicular skeletal
invasion culminating in a remarkable symptomatic improvement as a
result of radiotherapy administration.
Case Report
A 55-year-old Hispanic female presented with worsening lower
back pain, which radiated into her lower extremities bilaterally in July
2011. The patient reported being treated for similar symptoms over
the course of the previous year without relief from opioid or muscle
relaxer use. Although the patient reported pain and discomfort related
to muscle spasm, the patient had no neurologic deficits to suggest
severe spinal cord compression at any level at this point in her disease
course. However, upon review of previously obtained radiographic
images by her PCP, initial assessment was concerning for metastatic
lytic lesions of the spine with possible cord compression at T11-T12.
Follow-up imaging at the time of admission revealed an L4
compression fracture with severe spinal canal stenosis at L4-L5.
Multiple focal areas of intramedullary signal alteration were also
observed throughout the thoracic vertebrae, lumbar vertebrae, sacrum,
ilium and sternum. Such abnormalities are suspect for wide spread
metastasis from an unknown primary source or a form of skeletal
angiomatous disease. Subsequent CT guided needle core biopsy of the
left iliac wing yielded insufficient tissue and blood despite three
consecutive attempts. A repeat biopsy of the right sacrum revealed
bone with intramedullary vasculature suggestive of capillary type
Volume 6 • Issue 1 • 1000209
Citation:
Eisenbach C, Hansen C, Torres C (2015) The Role of Radiotherapy in the Treatment and Diagnosis of Pseudomalignant Vertebral
Haemangiomatosis: A Case Report. J Nucl Med Radiat Ther 6: 209. doi:10.4172/2155-9619.1000209
Page 2 of 5
hemangioma, essentially ruling out metastatic carcinoma of unknown
origin. In an effort to insure an accurate diagnosis, samples were sent
to Mayo Clinic and a confirmatory histopathology report of capillary
type hemangioma was later received. Despite subsequent patient
referral, problems encountered regarding patient insurance resulted in
a periodic lapse in care.
In March 2012, our patient again presented to her local emergency
department due to new onset, gradual loss of strength and paresthesias
in her lower extremities bilaterally for which she now required the
assistance of a walker to ambulate. Subsequent physical assessment
revealed marked atrophy of her right lower extremity musculature
with progressive weakness bilaterally. Considering the extent of
disease and rapid neurologic decline, the patient was immediately
referred for further imaging and a multi-disciplinary assessment to
develop a plan of care. An MRI revealed significant disease
progression with multiple enhancing lesions within the cervical spine,
clivus, right calvarium, right clavicular head and right humeral head.
Again, numerous enhancing lesions were visualized throughout the
thoracic vertebrae, lumbar vertebrae and sacrum with extension into
posterior elements i.e. pedicles, laminae and spinous processes.
Significant compression fractures of T5 and T11 compounded by
retropulsion of fragments into the spinal canal resulting in stenosis
and cord compression was noted. Furthermore, complete destruction
and collapse of L1 through L4 accompanied by compression of cauda
equina nerve roots was reported. Open biopsy on the heavily involved
L4 vertebral level was performed, followed by partial facetectomy,
pediculectomy and corpectomy in an attempt to partially relieve
compression and obtain another histologic sample. Histopathology of
the specimen was again indicative of extensive intraosseous capillary
type haemangioma. Following her recovery, the patient was informed
that due to the height of her disease burden, surgery was not a
possibility and she was discharged home.
Figure 1: (a-e) Sagittal and coronal magnetic resonance images of
cervical, thoracic and lumbar vertebral levels demonstrating
widespread tumor invasion and osseous destruction.1d :represents
a CT axial view demonstrating characteristic coarse bony
trabeculations, complete vertebral body involvement with pedicular
and transverse process invasion. Arrow heads represent areas of
spinal cord compression. Arrows represent intraosseous vertebral
involvement. Asterisks represent compression fractures.
Following several months of self-management and narcotic pain
control, the patient presented with symptomatic progression of total
motor paralysis and bilateral foot drop. Shortly thereafter, the patient
was referred to Southwest Cancer Treatment and Research Center in
J Nucl Med Radiat Ther
ISSN:2155-9619 JNMRT, an open access journal
Lubbock, Texas for assessment by a multi-disciplinary team of
neurosurgeons and radiation oncologists. A radiotherapy regimen was
decided upon and the patient received 30 GY in 15 fractions to her
thoracic vertebrae extending from T5 to T10. The treatment took place
from late May 2013 to mid-June 2013. Patient follow up in November
2013 revealed improvement in symptoms with the patient regaining
the ability to ambulate with a walker and eliminating her need for
narcotic pain medications. Six months post-radiotherapy, the patient
continued to improve from a symptomatic standpoint, with marked
improvement in her ability to ambulate requiring only intermittent
assistance from a cane. After 1 year post-radiotherapy, the patient
remains relatively asymptomatic with stable disease observed on MRI.
Discussion
Despite the relative commonality of vertebral haemangiomas, most
being found incidentally on imaging performed for unrelated
purposes, the vast majority of these benign endothelial
hamartomatous lesions are asymptomatic [3-5,9,21]. However, as in
the case of the patient presented above, significant discomfort and
neurologic decline has been documented in the literature, albeit very
rarely. Such symptoms include lower back pain, spasticity, weakness,
bladder and fecal incontinence, and paralysis to name a few [7-11].
Certain criteria in the literature aid in the recognition of potential or
frank neurologic deficits, many of which correlate directly to findings
on radiographic and nuclear imaging as seen in Table 1 [6,21-24].
From a pathophysiologic standpoint, categorization of primary
intraosseous haemangioma depends largely upon the vessel caliber and
degree of trabecular bone formation present within the lesion. Large
vessel formation and secondary dilation that prevents bony matrix
intrusion or fatty stromal invasion is indicative of cavernous
haemangiomas [21]. In contrast, small vessel endothelial proliferation
that permits development of bony trabeculae and stromal tissue
retention is descriptive of capillary-type haemangioma like that found
in the above patient. There is no denying that the pathophysiologic
process behind primary intraosseous haemangiomas can result in
cortical weakening and pathologic fracture, as seen in this patient. This
is likely due to a reactive bony sclerotic change that accompanies the
abnormal vascular proliferation. In addition, erosion into the pedicles,
laminae and spinous process harkens to a malignant process and is
unusual in vertebral haemangioma pathology [11,25,26]. Single, noninvasive, and non-destructive lesions are considered the norm in
regards to primary intraosseous haemangiomas. Thus, the undeniable
presence of, pathologic fracture, total vertebral invasion and
widespread involvement of the axial skeleton with extension into the
limbs, skull and pelvic girdle, in this patient represents an enigmatic
deviation from the norm. Ultimately, these facts altered the disease
trajectory and influenced therapeutic decision-making. Indeed, to
date, only a handful of individual cases in the literature describe
similar findings [11,25-27]. Yet, none, to our knowledge, have
demonstrated such extensive disease as that characterized by the
multiple compression fractures and multi-level spinal cord
compression points seen in this individual.
Since the first description of haemangiomas within the axial
skeleton in 1867, a tremendous effort has been put forth to
characterize and describe these lesions using radiographic and nuclear
imaging techniques [1]. Radiographically, vertebral haemangiomas
demonstrate a distinct pattern of prominent radiopaque trabeculae
distributed in a lamellar pattern interspersed with radiolucent
vasculature. Coined by Pernam in 1926, this characteristic ''jailhouse''
Volume 6 • Issue 1 • 1000209
Citation:
Eisenbach C, Hansen C, Torres C (2015) The Role of Radiotherapy in the Treatment and Diagnosis of Pseudomalignant Vertebral
Haemangiomatosis: A Case Report. J Nucl Med Radiat Ther 6: 209. doi:10.4172/2155-9619.1000209
Page 3 of 5
or ''honey-comb'' pattern along with any evidence of pathological
fracture should garner a high degree of suspicion for primary
intraosseous haemangioma when discovered on X-ray [5,6,21,28]. In
addition to traditional radiography, computed tomography scans have
emerged as a tremendously valuable imaging modality; providing
physicians with vital information regarding extent, invasion and
pathologic degenerative changes that may occur as a result of aberrant
stromal and vasculitic proliferation. Similar to its more primitive
cousin the traditional X-ray, transverse CT cross-sections of vertebral
haemangiomas yield a characteristic ''polka-dot'' appearance
representing the sclerotic columns of trabeculae surrounded by a large
amount of fatty stroma and prominent vasculature [6,21,29]. An
example of this phenomenon is evident in axial scan obtained from
this patient seen in Figure 1d. Despite the contributory role that the
above imaging techniques play in assessing osseous haemangiomas,
MRI remains the gold standard diagnostic imaging modality for axial
and appendicular skeletal haemangiomas revealing punctuate areas of
increased signal intensity in a background of decreased signal intensity
indicative, once again, of the sclerotic cancellous bone matrix within
fatty stroma and pronounced vasculature as seen in Figure 1a-1c and
1e [21,26,28]. All of the above findings are descriptive of simple benign
intraosseous hemangiomas. Yet, the specifics of the presented case
requires an in depth look at how the pseudomalignant, symptomatic,
invasive, and destructive primary intraosseous haemangiomas
subtypes differ from their asymptomatic, incidental counterparts upon
obtaining nuclear imaging. Fortunately, several anatomic features, if
observed on CT or MRI, have been found to be highly correlated with
aggressiveness, development of spinal cord compression, and severe
patient discomfort much like that observed in case presented above.
Likewise, certain absorbance patterns when obtained on nuclear
imaging are correlated with an increase in invasiveness [6,21,22,24,28].
For simplicity and clarity this information has been provided in the
following table:
Indicators for the Development/Presence of Symptomatic or Invasive Vertebral Haemangioma:
Nuclear Imaging and Anatomic Markers in Tumor Structure
Low signal intensity on T-1 weighted Magnetic Resonance Imaging
Evidence of expansion of haemangioma into the epidural space
Irregular ''honey-comb'' pattern on X-ray
Extension into posterior elements i.e. pedicles, lamina, neural arch
Complete vertebral body involvement
Variable post-contrast enhancement on CT or MRI
Hypodense mass with soft tissue invasion
Cortical expansion and pathologic fracture
Table 1: Pertinent imaging characteristics often observed in atypical and aggressive primary intraosseous haemangiomas.
Even though certain reliable symptomatic predictors exist, the
incidence of any symptom development in individuals with vertebral
haemangiomas is an extremely rare occurrence. However, in the case
of the above patient this usually benign tumor exhibited many of the
properties shown in Table 1 and has become one of only a few
documented cases of vertebral haemangiomatous disease in the
literature to demonstrate malignant characteristics from both a
nuclear imaging and symptomatic standpoint [7-11,25,28]. This
particular case demonstrates extremely rare and novel aspects of
primary intraosseous vertebral haemangiomas representing a distinct
subset of pathology that we have termed ''pseudomalignant
haemangiomatosis''. Considering the resemblance of this aggressive,
yet benign, primary intramedullary neoplastic subtype to lytic lesions
from a distant metastatic source it is essential to recognize the ''telltale''
signs of a haemangiomatous process. Namely, the ''jailhouse'' or
‘‘honeycomb” appearance on X-ray, the near pathognomonic ''polka
dot'' pattern on CT and the high signal intensity alteration on MRI
[6,21,22,24,28]. Such observations, when combined with clinical and
histologic evidence should aid in the successful recognition of a
statistical outlier like pseudomalignant haemangiomatosis.
Although it is well documented that a large majority of vertebral
haemangiomas require no treatment intervention. A clinical guideline
for the successful treatment of highly aggressive or symptomatic
vertebral haemangiomas has yet to be developed. Although no
consensus has been reached to justify one treatment modality over
another, it is important to keep in mind that disease presentation,
symptom management and rapidity of neurologic decline are essential
aspects to consider when deciding upon the therapeutic course of
action. For severe or recalcitrant pain or gradual neurologic decline,
radiotherapy has stood the test of time with multiple retrospective and
prospective studies demonstrating tremendous pain reduction, partial
to complete neurological deficit reversal, and decrease in overall
disease burden the details of which are graphically represented in
Table 2 [16,29-35]. With regards to this particular patient, this
information only serves to validate an already proven outcome.
Symptomatic Relief and Neurologic Recovery Obtained from Radiotherapy Administration in the Treatment of Vertebral Haemangioma
Primary Investigators
Number
Patients
Miszczyk and Ficek [30]
14
6 months
20-30 Gy
67%
33%
0%
Heyd [32]
84
68 months
34 Gy
61.9%
28.6%
9.5%
4.5 months
8-30 Gy
66.7%
33.3%
Suparna
[29]
and
Vadhiraja 6
of Average Follow -Up Average
Time
Dosage
J Nucl Med Radiat Ther
ISSN:2155-9619 JNMRT, an open access journal
Radiation Complete Pain Relief/ Partial Pain Relief/ No Pain Relief/No
Full
Neurologic Partial
Neurologic Neurologic Recovery
Recovery
Recovery
Volume 6 • Issue 1 • 1000209
Citation:
Eisenbach C, Hansen C, Torres C (2015) The Role of Radiotherapy in the Treatment and Diagnosis of Pseudomalignant Vertebral
Haemangiomatosis: A Case Report. J Nucl Med Radiat Ther 6: 209. doi:10.4172/2155-9619.1000209
Page 4 of 5
Yang and Zang [28]
23
2.5 months
30-40 Gy
80%
Asthana and Tandon [34]
17
1 month
35-40 Gy
76.4%
11.8%
11.8%
Aich and Deb [31]
7
6 months
40 Gy
100%
0%
0%
Templin et al. [17]
117
36-40 Gy
82%
Micke and Rades
82
70 months
34 Gy
61%
29.2%
9.8%
Faria [35]
9
8-44 months
30-40 Gy
88.9%
18 months
8-30 Gy
64%
Miszczk
[30]
and
Tukiendorf 101
11.1%
22%
14%
Table 2: Multiple prospective and retrospective studies demonstrating the success of radiotherapy in the treatment of symptomatic vertebral
haemangiomas.
Conclusion
In spite of the well documented, benign nature of this common
vascular abnormality, the variable presentation, atypical distribution
and extent of disease progression in this case represents a marked
detour regarding the so-called ''normal'' course of this tumor type. The
involvement of cervical, thoracic, lumbar, and sacral spinal segments
coupled with spread into the skull, right humerus and clavicle was
concerning for metastatic disease. Likewise, the invasion of posterior
vertebral elements resulting in total collapse, fragment retropulsion,
spinal canal stenosis and direct cord compression yielding significant
pain and lower extremity weakness again fostered a probable
metastatic etiology. Despite the presence of some ''typical'' nuclear
imaging findings, like that of the ''polka dot'' pattern on axial CT,
representing the coarse bony trabeculation amongst a background of
hypervascularity, the overwhelming aggressive characteristics observed
in this case may have played a role in delaying the ultimate diagnosis.
Overall, this case demonstrates the difficulty in differentiating between
the pseudomalignant variant of primary intraosseous haemangiomas
and a number of truly malignant bony neoplasms. In the midst of such
a rare and problematic diagnostic dilemma, there is no denying that
the therapeutic choice in this case brought about a complete
symptomatic turn around for the patient. This case solidifies the
essential role that directed, appropriately dosed, radiotherapy plays in
the treatment of even the most aggressive cases of primary
intraosseous vertebral haemangiomas.
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Haemangiomatosis: A Case Report. J Nucl Med Radiat Ther 6: 209. doi:10.4172/2155-9619.1000209
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